A rare case of adult ileo-colic intussusception: Hamartomatous polyp as a lead point with concurrent appendiceal neurofibroma in Neurofibromatosis Type 1


A rare case of adult ileo-colic intussusception: Hamartomatous polyp as a lead point with concurrent appendiceal neurofibroma in Neurofibromatosis Type 1


Sena Park, MD1*, Janaka Balasooriya, MBBS MD (Surgery) MRCS1

1Department of General Surgery, The Canberra Hospital, Yamba Drive, GARRAN, Australian Capital Territory 2605.


Intussusception is a rare cause of adult intestinal obstruction and unlike in children, adult intussusception is commonly caused by a lead point, requiring surgical intervention in most cases. Hamartomatous polyp is a non-neoplastic growth of tissue containing mature cells, distributed in an abnormal manner. It is often associated with intestinal polyposis syndromes such as Peutz-Jeghers syndrome and Juvenile polyposis. In the current case, we report an extremely rare case of ileo-colic intussusception secondary to a lead point of an isolated ileal hamartomatous polyp in an elderly gentleman with Neurofibromatosis type-1. Patient was successfully treated with ileo-colic resection involving the intussuscepted segment of bowel. There was an incidental finding of a nodule in the appendix and the histology confirmed this as a neurofibroma. Post-operative recovery of the patient was unremarkable.


Keywords: Intussusception; Hamartomatous polyp; Neurofibromatosis; Appendiceal neurofibroma


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How to cite this article:

Sena Park, Janaka Balasooriya. A rare case of adult ileo-colic intussusception: Hamartomatous polyp as a lead point with concurrent appendiceal neurofibroma in Neurofibromatosis Type 1. International Journal of Case Reports, 2021; 5:199. DOI: 10.28933/ijcr-2021-01-2505


References:

1. Gayer G, Apter S, Hofmann C, Nass S, Amitai M, Zissin R, Hertz M. Intussusception in adults: CT diagnosis. Clin Radiol. 1998;53(1):53-57. doi: 10.1016/s0009-9260(98)80035-4.
2. Azar T, Berger D. INTUSSUSCEPTION IN ADULTS. Annals of Surgery 1997;226(1):134-138.
3. Hanan B, Diniz TR, da Luz MM, da Conceição SA, da Silva RG, Lacerda-Filho A. Intussusception in adults: a retrospective study. Colorectal Dis. 2010;12(6):574-578. doi: 10.1111/j.1463-1318.2009.01865.x.
4. Jelsig AM. Hamartomatous polyps – a clinical and molecular genetic study. Dan Med J. 2016;63(8):B5280.
5. Ozer A, Sarkut P, Ozturk E, Yilmazlar T. Jeju-noduodenal intussusception caused by a soli-tary polyp in a woman with Peutz-Jeghers syn-drome: a case report. Journal of Medical Case Reports 2014;8(1):13.
6. Yalamarthi S, Smith R. Adult intussusception: case reports and review of literature. Post-graduate Medical Journal 2005;81:174-177.
7. Adebamowo CA, Akang EE, Pindiga HU, Ezeome ER, Omotosho PO, Labeodan OA, Solanke TF. Changing clinicopathological profile of intussusception in Nigeria–a 20-year review. Hepatogastroenterology. 2000;47(32):437-440.
8. Marinis A, Yiallourou A, Samanides L, Dafnios N, Anastasopoulos G, Vassiliou I, et al. Intussus-ception of the bowel in adults: a review. World Journal of Gastroenterology 2009;15(4):407-411.
9. Hong KD, Kim J, Ji W, Wexner SD. Adult in-tussusception: a systemic review and me-ta-analysis. Techniques in Coloproctology 2019;23 (1):315-324.
10. Valentini V, Buquicchio GL, Galluzzo M, Ianniello S, Di Grezia G, Ambrosio R, Trinci M, Miele V. Intussusception in Adults: The Role of MDCT in the Identification of the Site and Cause of Ob-struction. Gastroenterol Res Pract. 2016;2016: 5623718. doi: 10.1155/2016/5623718
11. Boulier K, Erwin DJ, Nagamani S, Eble TN. A case report of hamartomatous polyposis in an individual with Neurofibromatosis type 1. Clin Case Rep. 2018 Dec 11;7(1):202-205. doi: 10.1002/ccr3.1908
12. Brosens LA, Offerhaus GJ, Canto MI, Mont-gomery EA, Giardiello FM. Simultaneous juve-nile polyposis syndrome and neurofibromatosis type 1. Histopathology. 2016 Jan;68(2):313-5. doi: 10.1111/his.12734. Epub 2015 Jun 18. PMID: 25951773; PMCID: PMC4636475.
13. Komo T, Oishi K, Kohashi T, Hihara J, Yo-shimitsu M, Tokumoto N, et al. Appendiceal neurofibroma with low-grade appendiceal mu-cinous neoplasm in neurofibromatosis type 1 patient: A case report. International journal of surgery case reports 2018;53:377-380.
14. Agaimy A, Schaefer I, Kotzina L, Knolle J, Baumann I, Ströbel P, et al. Juvenile-like (in-flammatory/hyperplastic) mucosal polyps of the gastrointestinal tract in neurofibromatosis type 1. Histopathology 2014 05/01; 64(6):777-786.
15. Oktenli C, Gul D, Deveci MS, Saglam M, Upadhyaya M, Thompson P, et al. Unusual features in a patient with neurofibromatosis type 1: Multiple subcutaneous lipomas, a juvenile polyp in ascending colon, congenital intrahe-patic portosystemic venous shunt, and horse-shoe kidney. Am J Med Genet 2004 06/15; 2021/01;127A(3):298-301.
16. Brosens LA, Offerhaus GJ, Canto MI, Mont-gomery EA, Giardiello FM. Simultaneous juve-nile polyposis syndrome and neurofibromatosis type 1. Histopathology. 2016;68(2):313-315. doi:10. 1111/his.12734
17. Oktenli C, Gul D, Deveci MS, Saglam M, Upadhyaya M, Thompson P, et al. Unusual features in a patient with neurofibromatosis type 1: Multiple subcutaneous lipomas, a juvenile polyp in ascending colon, congenital intrahe-patic portosystemic venous shunt, and horse-shoe kidney. Am J Med Genet 2004 06/15; 2021/01;127A(3):298-301.
18. Tan KY, Tan SM, Tan AG, Chen CY, Chng HC, Hoe MN. Adult intussusception: experience in Singapore. ANZ J Surg. 2003;73(12):1044-7. doi: 10.1046/j.1445-2197.2003.t01-22-.x.
19. Begos DG, Sandor A, Modlin IM. The diagnosis and management of adult intussusception. Am J Surg. 1997;173(2):88-94. doi: 10.1016/S0002-9610(96)00419-9.
20. Tan KY, Tan SM, Tan AG, Chen CY, Chng HC, Hoe MN. Adult intussusception: experience in Singapore. ANZ J Surg. 2003;73(12):1044-7. doi: 10.1046/j.1445-2197.2003.t01-22-.x.